Bernasconi, E.

Huber, X.

Rosso, R.

Tschuor-Costa, S.

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CASE REPORTExceptional dissemination of perineal tuberculosis up to the rightflank: a tribute to J.P. Nesselrod’s study on the anatomy of pelviclymphatics published in 1936 in the Annals of SurgeryX. Huber & E. Bernasconi & S. Tschuor-Costa & R. RossoAccepted: 27 November 2012 /Published online: 19 January 2013# The Author(s) 2013. This article is published with open access at Springerlink.comCaseA 56-year-old cachectic male in poor general and hygieniccondition collapsed and was subsequently admitted to hos-pital in a severe sepsis. The personal history was negativefor preexisting intraabdominal and cardiorespiratory ail-ments. Recent journeys to tropical countries were denied.However, a lengthy stay at a psychiatric clinic due to alcoholabuse several years ago was noteworthy. The patient hadbeen suffering from perineal fistula for years without anymedical treatment. At admission, we found a complex ofperianal and gluteal abscesses with involvement of the skinof the right lower back. The abdomen was clinically unsus-picious. The laboratory studies revealed a severe inflamma-tory syndrome measuring a white cell count of 30×109/L,CRP value of 226 mg/L, and slightly elevated procalcito-nine with 0.47 μg/L.A CT scan revealed an extensive subcutaneous abscesson the right flank with involvement of the right iliopsoasmuscle (Fig. 1), the presacral space, the fossa ischiorectaliswith involvement of the external sphincter, and the levatorani (Fig. 2a–c) with subcutaneous fistula in the perianalregion and the adjacent right gluteus. However, no internalorifice was found by rigid rectoscopy. An endoanal ultra-sound to investigate the presence of trans- or intersphinc-teric fistula was omitted due to the patient's bad condition.The MRI performed to analyze whether there was an in-volvement of vertebral bodies or an involvement of inter-vertebral disks was not suitable to evaluate the analsphincter.There were no particular intraabdominal findings such assignificant lymphatic nodes, wall thickening of the bowel,or ascites. All findings were restricted to the extraperitonealspace.Despite immediate surgical drainage of the subcutaneousabscess on the right flank and in the perianal region as wellas antibiotic treatment with piperacilline/tazobactam, thepatient's condition showed signs of deterioration such asweight loss, hypotension, and fever. Additionally, upcomingperianal abscesses were surgically evacuated, and the pre-sacral space was partially drained initially by CT-guidedpuncture and finally emptied by open surgery over anextraperitoneal right–anterior access to the right iliopsoasmuscle. The drained perianal wounds did not show anysigns of healing and continued to produce serous turbidsecretion. Crohn's disease was suspected early, but not con-firmed in an ileocolonoscopy. The microbial culture fromR. RossoDepartment of General Surgery,Ospedale Regionale di Lugano, Via Tesserete 46,CH-6900 Lugano, SwitzerlandE. BernasconiDepartment of Infectious Diseases,Ospedale Regionale di Lugano, Via Tesserete 46,CH-6900 Lugano, SwitzerlandS. Tschuor-CostaDepartment of Radiology,Ospedale Regionale di Lugano, Via Tesserete 46,CH-6900 Lugano, SwitzerlandX. HuberDepartment of Surgery, University Hospital Basel,Spitalstrasse 31,CH-4031 Basel, SwitzerlandX. Huber (*)Department of General Surgery, University Hospital,Spitalstrasse 31,CH-4031 Basel, Switzerlande-mail: Xaver.Huber@usb.chR. Rosso (*)Department of Surgery, Ospedale Regionale, Via Tesserete 46,CH-6903 Lugano, Switzerlande-mail: raffaele.rosso@eoc.chInt J Colorectal Dis (2013) 28:723–726DOI 10.1007/s00384-012-1624-2the wound secretion demonstrated growth of Streptococcusintermedius, whereas the surgically obtained specimensfrom the right presacral abscess evolved to be sterile. Sero-logical screening for syphilis and HIV was negative. In ahistological sample from the perineal region, tuberculoidgranuloma was found, while an interferon gamma releaseassay (Quantiferon Gold) in the blood was positive. Thepatient did not suffer from any pulmonary symptoms, but aCT scan of the chest showed reticulonodular infiltrates typ-ically suggestive of pulmonary tuberculosis. Microscopy ofthe sputum was negative for acid-fast bacilli, but the cultureof surgical probes from the perineum and the retroperitonealspace finally became positive for Mycobacterium tubercu-losis about 1 month after admission. The patient was pre-scribed a classic tuberculostatic treatment combiningrifampin, isoniazid, pyrazinamide, and ethambutol. We ob-served a rapid healing of the wounds, a significant increaseof the patient's body weight, and recovery of the generalcondition.DiscussionIt is estimated that abdominal tuberculosis accounts forabout 1 % of all extrapulmonary manifestations of tubercu-losis (TB) [1, 2]. However, the true rate of abdominalinvolvement might be underestimated [3, 4]. Perianal tuber-culosis is reported to be rare and mainly referred to aschronic fistula formation or ulceration related to pulmonaryTB [1, 5]. In one series of anal sepsis, TB was found inabout 1 % of all cases [5]. In everyday life, the generalsurgeon is often confronted with perineal abscesses causedby common bacteria of the intestinal tract. In more devel-oped countries, complex fistula formation is mainly sugges-tive of Crohn's disease that mimics TB in the abdominalcavity as well as in the perianal region [3]. Abdominal TBmost often appears with abdominal pain and distension,weight loss, anorexia, and fever. Intestinal bleeding due toulceration, intestinal perforation, or obstruction are possiblecomplications that urge a surgical intervention. The ileum,the ileocecal region (including mesenteric lymph nodes),and the peritoneum are found to be involved most frequently[6, 7]. Computed tomography findings correlate with theclinical findings: wall thickening of the small bowel, mes-enteric lymphadenopathy, and ascites are usually reportedfrom patients with abdominal TB [3, 8].In our case, an evident involvement of abdominal organswas absent. The patient never had abdominal discomfort,the ileocolonoscopy was normal, and in the CT scan, signsof intestinal or peritoneal involvement were absent.Anatomically, the extension of the disease was restrictedto the right iliopsoas muscle and surrounding presacraltissues, as well as to the perianal region. Although werepeatedly examined the patient clinically, we were not ableto find any fistula passing the anal sphincter. However, wea b cFig. 2 Coronary reconstruction of the patients' CT scan on the level ofthe femur (f) and os ischium (oi). Panel a shows a subcutaneous fistula(arrow) reaching the levator ani (triangles). In b, the subcutaneousabscess (star) is linked over a fistula (arrow) with the skin. In c, thepresacral space is filled with abscess (star) whereas bilateral lymphnodes are visible (triangles)Fig. 1 The patients' CT scan at admission, showing a large subcuta-neous abscess (star) on the flank at the level of the fourth lumbarvertebral body, communicating with the iliopsoas muscle, the para-spinal muscles, and the epidural space (arrow)724 Int J Colorectal Dis (2013) 28:723–726were astonished at the bizarre subcutaneous abscess formationon the right flank that was the leading finding at admission toour hospital. An initially suspected spondylodiscitis could notbe confirmed by repeatedMRI scans. Overall, we interpret thepresented case of perianal tuberculosis as a hematogenousdissemination of pulmonary tuberculosis.Numerous studies have evaluated the practical conse-quences of perianal lymphatic drainage in the context ofmalignant disease. It is well established that tumors belowthe linea dentata drain to the inguinal lymph nodes [9–11].Our patient showed no significant inguinal lymphadenopa-thy, neither in the clinical exam nor in the repeated CT scan.a bFig. 3 Sagittal reconstructions of the patient's CT scan at admission.On a, the subcutaneous abscess (star) and its breakthrough to theiliopsoas muscle (triangle) over the right crista iliaca (il os ilium) areshown. A thin subcutaneous spur (arrows) delineates potential routesof disease expansion (b) on the level of the lumbar vertebral column(L5 fifth lumbar vertebral body, OS os sacrum)a bFig. 4 Original figure from Nesselrod's publication in 1936 [12] inAnnals of Surgery showing the perineal and gluteal distribution of lym-phatic vessels after injections of mercury in the skin, respectively. Panel ashows a dorsal view of the sacrococcygeal and superior gluteal region of amale white fetus at term (original description). Perianal (e), presacral (b),gluteal plexuses (a) and anastomotic vessels (d). Panel b shows a lateralview on the same specimen as in a, showing not only lymphatic vesselsabove the crista iliaca but also their link (c) to the inguinal zoneInt J Colorectal Dis (2013) 28:723–726 725But the iliacal axis was certainly involved (Figs. 2c and 3a).While searching for an explanation for the exceptional ex-tension and localisation of the inflammatory process involv-ing the subcutaneous tissue on the right flank and the rightiliopsoas muscle, we came across an anatomic study pub-lished by J.P. Nesselrod in 1936 ([12]; Fig. 4).The eminent proctologist revealed the existence of anas-tomotic lymphatic vessels between the perianal skin and aspecific gluteal plexus over the buttocks and further back-wards to the flank above the crista iliaca. Nesselrod con-ducted studies with cadavers of human fetuses at or nearterm, of newborn infants, as well as few but apparently notuseful studies on dogs and humans in vivo. He used differ-ent dyes, whereby the best and depicted results (Fig. 4) wereachieved with mercury which had been injected in theperianal skin. As shown in Fig. 4a, there is a perianalnetwork linked with another network over the crista iliacaover gluteal and sacrococcygeal plexuses. Over the supra-iliacal plexuses, there is a continuing connection to theinguinal nodes hereby encircling the limb. Nesselrod alsodescribes some minor anastomotic lymphatic vessels thatpass the anorectal line and ascend in the columns of Mor-gagni in the ampulla recti. Unfortunately, he does not statewhether the iliacal axis was involved upon injection in theperianal plexus.It is astonishing per se that such an abscess lies subcuta-neously on the right flank. This is rare and requires anexplanation. Since it was a case of secondary tuberculosis,it must have been a consequence of hematogenous dissem-ination and not, as it was possible in the case of trueintestinal tuberculosis, as a primary infection of the intes-tine. Principally, it could have been a direct seeding into theright flank. It has to be underscored that the most frequentextrapulmonary manifestation of tuberculosis is the lym-phatic form [13]. Therefore, lymphatic networks representan anatomic space of preferential “homing” for tuberculosis.The sheer presence of lymphatic vessels or plexuses abovethe crista iliaca made the manner of TB seeding and survivalat that precise location more plausible.Of course, the direction of disease dissemination (break-through through the abdominal flank into or out from theiliopsoas muscle) cannot be definitively cleared. Nesselrodhimself reports that perianally injected dyes rise up to thebuttocks, but can also descend from the buttocks to the peria-nal region. Consequently, it is a bidirectional system. Becauseof anamnestic hints, one has to assume that the primary focuswas a perianal abscess that probably took both routes. In ourcase, there were minor subcutaneous fistulas forming spurs upto the right gluteus (Fig. 3a and b). Internally, CT scan pro-vides clear evidence of the involvement of the puborectaliszone and the iliacal axis (Fig. 2). The iliacal axis only couldhave been reached over the involvement of the puborectaliszone. What Nesselrod does not show are anastomotic vesselsthrough the abdominal wall. So it seems reasonable to assumethat there were two ways of dissemination (an external and aninternal one) that completed an anatomic circle and broke atrue anatomic barrier by passing the fascia scarpa and con-necting the plexuses above the crista iliaca and the lymphaticsof the iliacal axis.Correlating these old studies with our anatomic findings,we see the clinical relevance of these lymphatic vessels onthe buttocks by their concrete involvement.Acknowledgments The authors thank N. Jonkers for languagecorrection.Conflict of interest None.Open Access This article is distributed under the terms of the Crea-tive Commons Attribution License which permits any use, distribution,and reproduction in any medium, provided the original author(s) andthe source are credited.References1. Tai WC, Hu TH, Ch L et al (2010) Ano-perianal tuberculosis:15 years of clinical experiences in Southern Taiwan. Colorectal Dis12(7 Online):e120–e2242. Barker JA, Conway AM, Hill J (2011) Supralevator fistula-in-anoin tuberculosis. Color Dis 13(2):210–2143. Almadi MA, Ghosh S, Aljebreen AM (2009) Differentiating intes-tinal tuberculosis from Crohn's disease: a diagnostic challenge. AmJ Gastroenterol 104(4):1003–10124. Aston NO (1997) Abdominal tuberculosis. World J Surg 21(5):492–4995. Kraemer M, Gill SS, Seow-Choen F (2000) Tuberculous analsepsis: report of clinical features in 20 cases. Dis Colon Rectum43(11):1589–15916. Hassan I, Brilakis ES, Thompson RL et al (2002) Surgical man-agement of abdominal tuberculosis. J Gastrointest Surg 6(6):862–8677. Donoghue HD, Holton J (2009) Intestinal tuberculosis. Curr OpinInfect Dis 22(5):490–4968. Tan KK, Chen K, Sim R (2009) The spectrum of abdominaltuberculosis in a developed country: a single institution's experi-ence over 7 years. J Gastrointest Surg 13(1):142–1479. Frost DB, Richards PC, Montague ED et al (1984) Epidermoidcancer of the anorectum. Cancer 53(6):1285–8510. Greenali MJ, Quan SH, Stearns MW et al (1985) Epidermoidcancer of the anal margin. Pathologic features, treatment andclinical results. Am J Surg 149(1):95–10111. Ryan DP, Compton CC, Mayer RJ (2000) Carcinoma of the analcanal. N Engl J Med 342(11):792–80012. Nesselrod JP (1936) An anatomic restudy of the pelvic lymphatics.Ann Surg 104(5):905–91613. Mehta JB, Dutt A, Harvill L, Mathews KM (1991) Epidemiologyof extrapulmonary tuberculosis. A comparative analysis with pre-AIDS era. Chest 99:1134–1138726 Int J Colorectal Dis (2013) 28:723–726

Exceptional dissemination of perineal tuberculosis up to the right flank: a tribute to J.P. Nesselrod's study on the anatomy of pelvic lymphatics published in 1936 in the Annals of Surgery

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Exceptional dissemination of perineal tuberculosis up to the right flank: a tribute to J.P. Nesselrod's study on the anatomy of pelvic lymphatics published in 1936 in the Annals of Surgery

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